A 33-year-old Belgian woman was referred in June 2006 to our Metabolic Bone Diseases Clinic for recent vertebral fractures. X-Rays showed a 3-level severe compression fractures with collapse of the superior endplates of D11, D12 and L1. DXA showed significantly decreased lumbar spine (LS) and femoral neck (FN) Z-scores, at −3.1 and −2.5, respectively.
She was complaining of severe back pain, muscle weakness, cognitive dysfunction, anxiety and irritability. Physical examination revealed moon-shaped face, ecchymosis, purple stretch marks on the breasts, arms, abdomen and thighs.
Morning cortisol was normal but ACTH level was suppressed. Urinary free cortisol was elevated. A late-night salivary cortisol also elevated. Standard 2-day 2-mg dexamethasone suppression test showed no cortisol suppression. A loss of diurnal variation in ACTH and cortisol levels was also demonstrated. Cushings syndrome was confirmed. Abdominal MRI showed a 3 cm left adrenal mass.
Osteoporosis was considered secondary to this endogenous hypercortisolism. Bone turnover markers (BTMs) and 24h urinary calcium excretion were elevated. Successful laparoscopic left adrenalectomy was performed in October 2006. Spontaneous BMD increase (LS Z-score −2.4) was observed already 2 months after surgery. Except for calcium and vitamin D supplementation, no bisphosphonate therapy was prescribed, given this impressive increase of BMD and because she was still premenopausal. X-Rays did not show any new vertebral fracture. BMD continued to increase. 24 months after surgery, densitometry showed 40% absolute increase in LS BMD and 16% in FN BMD, vs baseline. She reached normal BMD values at all sites 3 years following surgery.
After a 9-years follow-up (DXA and BTMs, yearly), complete spontaneous BMD recovery was confirmed (LS T-score +1.2 and FN T-score −0.3) with normal and stable BTMs.
Our case suggests no need for any antiresorptive therapy in severe osteoporosis due to cortisol-secreting adrenal adenoma after successful surgery, at least in premenopausal women.
20 - 23 May 2017
European Society of Endocrinology